SynthesisNeurology(R) neuroimmunology & neuroinflammation2024
Defining the Clinicoradiologic Syndrome of SARS-CoV-2 Acute Necrotizing Encephalopathy: A Systematic Review and 3 New Pediatric Cases
Open access · goldAbstract readSystematic Review
In one paragraph
Synthesis in Neurology(R) neuroimmunology & neuroinflammation, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.
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0cells of the map it votes in
7citing papers in PubMed
1.9field-weighted citation impact, top 13% of its field
What it found
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Who cites it
7 citing papers in PubMed, 9 citations in OpenAlex.
- Infection-triggered encephalopathy syndromes: a meta-analysis of clinical characteristics and outcomes in 1946 cases.EClinicalMedicine · 2026Article
- Acute necrotizing encephalopathy associated with COVID-19 in a pediatric patient and a systematic review.Journal of human immunity · 2026Article
- High-dose methylprednisolone and tocilizumab improve survival of patients with high-risk pediatric acute necrotizing encephalopathy.Clinical and experimental pediatrics · 2026Article
- Complications of Therapeutic Plasma Exchange in Pediatric Neuroimmune Disorders.Children (Basel, Switzerland) · 2025Article
- International consensus definitions for infection-triggered encephalopathy syndromes.Developmental medicine and child neurology · 2025Article
- Predictive value of ANE-SS combined with ferritin and DIC scores for mortality Risk in children with acute necrotizing encephalopathy.Translational pediatrics · 2025Article
- Acute necrotizing encephalopathy caused by bacterial infection.BMC infectious diseases · 2024Article
Corrections and comments
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Authors and funding
10 authors at 1 institution in 6 countries.
Vanessa W LeeFrom the Children's Neurosciences (V.W.L., M.L.), Evelina London Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre; Infectious Disease Service (K.Q.K.), Department of Paediatrics, KK Women's and Children's Hospital; SingHealth Duke-NUS Paediatrics Academic Clinical Program (ACP) (K.Q.K., T.T.), KK Women's and Children's Hospital, Singapore; Paediatric Neurology Unit (A.R.M., H.M., P.A.), Hospital Tunku Azizah, Kuala Lumpur; Department of Paediatrics (H.M.), Faculty of Medicine and Health Sciences, Universiti Putra Malaysia, Serdang; Department of Immunology (Q.S.), School of Basic Medical Sciences, Fujian Medical University; Department of Obstetrics (Q.S.), Fujian Maternity and Child Health Hospital, Fuzhou, China; Department of Biochemistry (A.F.P.), University of Toronto, Ontario, Canada; Kids Neuroscience Centre (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney; Clinical School (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney, New South Wales, Australia; Department Women and Children's Health (M.L.), School of Life Course Sciences (SoLCS), King's College London, United Kingdom; and Neurology Service (T.T.), Department of Paediatrics, KK Women's and Children's Hospital, Singapore.ORCID 0000-0003-3988-204X
Kai Qian KamFrom the Children's Neurosciences (V.W.L., M.L.), Evelina London Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre; Infectious Disease Service (K.Q.K.), Department of Paediatrics, KK Women's and Children's Hospital; SingHealth Duke-NUS Paediatrics Academic Clinical Program (ACP) (K.Q.K., T.T.), KK Women's and Children's Hospital, Singapore; Paediatric Neurology Unit (A.R.M., H.M., P.A.), Hospital Tunku Azizah, Kuala Lumpur; Department of Paediatrics (H.M.), Faculty of Medicine and Health Sciences, Universiti Putra Malaysia, Serdang; Department of Immunology (Q.S.), School of Basic Medical Sciences, Fujian Medical University; Department of Obstetrics (Q.S.), Fujian Maternity and Child Health Hospital, Fuzhou, China; Department of Biochemistry (A.F.P.), University of Toronto, Ontario, Canada; Kids Neuroscience Centre (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney; Clinical School (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney, New South Wales, Australia; Department Women and Children's Health (M.L.), School of Life Course Sciences (SoLCS), King's College London, United Kingdom; and Neurology Service (T.T.), Department of Paediatrics, KK Women's and Children's Hospital, Singapore.ORCID 0000-0002-2402-7262
Ahmad R MohamedFrom the Children's Neurosciences (V.W.L., M.L.), Evelina London Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre; Infectious Disease Service (K.Q.K.), Department of Paediatrics, KK Women's and Children's Hospital; SingHealth Duke-NUS Paediatrics Academic Clinical Program (ACP) (K.Q.K., T.T.), KK Women's and Children's Hospital, Singapore; Paediatric Neurology Unit (A.R.M., H.M., P.A.), Hospital Tunku Azizah, Kuala Lumpur; Department of Paediatrics (H.M.), Faculty of Medicine and Health Sciences, Universiti Putra Malaysia, Serdang; Department of Immunology (Q.S.), School of Basic Medical Sciences, Fujian Medical University; Department of Obstetrics (Q.S.), Fujian Maternity and Child Health Hospital, Fuzhou, China; Department of Biochemistry (A.F.P.), University of Toronto, Ontario, Canada; Kids Neuroscience Centre (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney; Clinical School (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney, New South Wales, Australia; Department Women and Children's Health (M.L.), School of Life Course Sciences (SoLCS), King's College London, United Kingdom; and Neurology Service (T.T.), Department of Paediatrics, KK Women's and Children's Hospital, Singapore.ORCID 0000-0002-3838-923X
Husna MusaFrom the Children's Neurosciences (V.W.L., M.L.), Evelina London Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre; Infectious Disease Service (K.Q.K.), Department of Paediatrics, KK Women's and Children's Hospital; SingHealth Duke-NUS Paediatrics Academic Clinical Program (ACP) (K.Q.K., T.T.), KK Women's and Children's Hospital, Singapore; Paediatric Neurology Unit (A.R.M., H.M., P.A.), Hospital Tunku Azizah, Kuala Lumpur; Department of Paediatrics (H.M.), Faculty of Medicine and Health Sciences, Universiti Putra Malaysia, Serdang; Department of Immunology (Q.S.), School of Basic Medical Sciences, Fujian Medical University; Department of Obstetrics (Q.S.), Fujian Maternity and Child Health Hospital, Fuzhou, China; Department of Biochemistry (A.F.P.), University of Toronto, Ontario, Canada; Kids Neuroscience Centre (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney; Clinical School (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney, New South Wales, Australia; Department Women and Children's Health (M.L.), School of Life Course Sciences (SoLCS), King's College London, United Kingdom; and Neurology Service (T.T.), Department of Paediatrics, KK Women's and Children's Hospital, Singapore.ORCID 0000-0002-0784-2302
Poorani AnandakrishnanFrom the Children's Neurosciences (V.W.L., M.L.), Evelina London Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre; Infectious Disease Service (K.Q.K.), Department of Paediatrics, KK Women's and Children's Hospital; SingHealth Duke-NUS Paediatrics Academic Clinical Program (ACP) (K.Q.K., T.T.), KK Women's and Children's Hospital, Singapore; Paediatric Neurology Unit (A.R.M., H.M., P.A.), Hospital Tunku Azizah, Kuala Lumpur; Department of Paediatrics (H.M.), Faculty of Medicine and Health Sciences, Universiti Putra Malaysia, Serdang; Department of Immunology (Q.S.), School of Basic Medical Sciences, Fujian Medical University; Department of Obstetrics (Q.S.), Fujian Maternity and Child Health Hospital, Fuzhou, China; Department of Biochemistry (A.F.P.), University of Toronto, Ontario, Canada; Kids Neuroscience Centre (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney; Clinical School (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney, New South Wales, Australia; Department Women and Children's Health (M.L.), School of Life Course Sciences (SoLCS), King's College London, United Kingdom; and Neurology Service (T.T.), Department of Paediatrics, KK Women's and Children's Hospital, Singapore.ORCID 0000-0002-2357-2074
Qingtang ShenFrom the Children's Neurosciences (V.W.L., M.L.), Evelina London Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre; Infectious Disease Service (K.Q.K.), Department of Paediatrics, KK Women's and Children's Hospital; SingHealth Duke-NUS Paediatrics Academic Clinical Program (ACP) (K.Q.K., T.T.), KK Women's and Children's Hospital, Singapore; Paediatric Neurology Unit (A.R.M., H.M., P.A.), Hospital Tunku Azizah, Kuala Lumpur; Department of Paediatrics (H.M.), Faculty of Medicine and Health Sciences, Universiti Putra Malaysia, Serdang; Department of Immunology (Q.S.), School of Basic Medical Sciences, Fujian Medical University; Department of Obstetrics (Q.S.), Fujian Maternity and Child Health Hospital, Fuzhou, China; Department of Biochemistry (A.F.P.), University of Toronto, Ontario, Canada; Kids Neuroscience Centre (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney; Clinical School (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney, New South Wales, Australia; Department Women and Children's Health (M.L.), School of Life Course Sciences (SoLCS), King's College London, United Kingdom; and Neurology Service (T.T.), Department of Paediatrics, KK Women's and Children's Hospital, Singapore.ORCID 0000-0001-5520-8480
Alexander F PalazzoFrom the Children's Neurosciences (V.W.L., M.L.), Evelina London Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre; Infectious Disease Service (K.Q.K.), Department of Paediatrics, KK Women's and Children's Hospital; SingHealth Duke-NUS Paediatrics Academic Clinical Program (ACP) (K.Q.K., T.T.), KK Women's and Children's Hospital, Singapore; Paediatric Neurology Unit (A.R.M., H.M., P.A.), Hospital Tunku Azizah, Kuala Lumpur; Department of Paediatrics (H.M.), Faculty of Medicine and Health Sciences, Universiti Putra Malaysia, Serdang; Department of Immunology (Q.S.), School of Basic Medical Sciences, Fujian Medical University; Department of Obstetrics (Q.S.), Fujian Maternity and Child Health Hospital, Fuzhou, China; Department of Biochemistry (A.F.P.), University of Toronto, Ontario, Canada; Kids Neuroscience Centre (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney; Clinical School (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney, New South Wales, Australia; Department Women and Children's Health (M.L.), School of Life Course Sciences (SoLCS), King's College London, United Kingdom; and Neurology Service (T.T.), Department of Paediatrics, KK Women's and Children's Hospital, Singapore.ORCID 0000-0002-9700-1995
Russell C DaleFrom the Children's Neurosciences (V.W.L., M.L.), Evelina London Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre; Infectious Disease Service (K.Q.K.), Department of Paediatrics, KK Women's and Children's Hospital; SingHealth Duke-NUS Paediatrics Academic Clinical Program (ACP) (K.Q.K., T.T.), KK Women's and Children's Hospital, Singapore; Paediatric Neurology Unit (A.R.M., H.M., P.A.), Hospital Tunku Azizah, Kuala Lumpur; Department of Paediatrics (H.M.), Faculty of Medicine and Health Sciences, Universiti Putra Malaysia, Serdang; Department of Immunology (Q.S.), School of Basic Medical Sciences, Fujian Medical University; Department of Obstetrics (Q.S.), Fujian Maternity and Child Health Hospital, Fuzhou, China; Department of Biochemistry (A.F.P.), University of Toronto, Ontario, Canada; Kids Neuroscience Centre (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney; Clinical School (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney, New South Wales, Australia; Department Women and Children's Health (M.L.), School of Life Course Sciences (SoLCS), King's College London, United Kingdom; and Neurology Service (T.T.), Department of Paediatrics, KK Women's and Children's Hospital, Singapore.ORCID 0000-0002-2495-1826
Ming LimFrom the Children's Neurosciences (V.W.L., M.L.), Evelina London Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre; Infectious Disease Service (K.Q.K.), Department of Paediatrics, KK Women's and Children's Hospital; SingHealth Duke-NUS Paediatrics Academic Clinical Program (ACP) (K.Q.K., T.T.), KK Women's and Children's Hospital, Singapore; Paediatric Neurology Unit (A.R.M., H.M., P.A.), Hospital Tunku Azizah, Kuala Lumpur; Department of Paediatrics (H.M.), Faculty of Medicine and Health Sciences, Universiti Putra Malaysia, Serdang; Department of Immunology (Q.S.), School of Basic Medical Sciences, Fujian Medical University; Department of Obstetrics (Q.S.), Fujian Maternity and Child Health Hospital, Fuzhou, China; Department of Biochemistry (A.F.P.), University of Toronto, Ontario, Canada; Kids Neuroscience Centre (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney; Clinical School (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney, New South Wales, Australia; Department Women and Children's Health (M.L.), School of Life Course Sciences (SoLCS), King's College London, United Kingdom; and Neurology Service (T.T.), Department of Paediatrics, KK Women's and Children's Hospital, Singapore.ORCID 0000-0001-7738-8910
Terrence ThomasFrom the Children's Neurosciences (V.W.L., M.L.), Evelina London Children's Hospital at Guy's and St Thomas' NHS Foundation Trust, King's Health Partners Academic Health Science Centre; Infectious Disease Service (K.Q.K.), Department of Paediatrics, KK Women's and Children's Hospital; SingHealth Duke-NUS Paediatrics Academic Clinical Program (ACP) (K.Q.K., T.T.), KK Women's and Children's Hospital, Singapore; Paediatric Neurology Unit (A.R.M., H.M., P.A.), Hospital Tunku Azizah, Kuala Lumpur; Department of Paediatrics (H.M.), Faculty of Medicine and Health Sciences, Universiti Putra Malaysia, Serdang; Department of Immunology (Q.S.), School of Basic Medical Sciences, Fujian Medical University; Department of Obstetrics (Q.S.), Fujian Maternity and Child Health Hospital, Fuzhou, China; Department of Biochemistry (A.F.P.), University of Toronto, Ontario, Canada; Kids Neuroscience Centre (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney; Clinical School (R.C.D.), The Children's Hospital at Westmead, Faculty of Medicine and Health, University of Sydney, New South Wales, Australia; Department Women and Children's Health (M.L.), School of Life Course Sciences (SoLCS), King's College London, United Kingdom; and Neurology Service (T.T.), Department of Paediatrics, KK Women's and Children's Hospital, Singapore.ORCID 0000-0003-4241-1652
The University of Sydney · AU
Funding
No grant is acknowledged in the PubMed record.
Abstract
BACKGROUND AND
objectivesWe characterize clinical and neuroimaging features of SARS-CoV-2-related acute necrotizing encephalopathy (ANE).
methodsSystematic review of English language publications in PubMed and reference lists between January 1, 2020, and June 30, 2023, in accordance with PRISMA guidelines. Patients with SARS-CoV-2 infection who fulfilled diagnostic criteria for sporadic and genetic ANE were included.
resultsFrom 899 articles, 20 cases (17 single case reports and 3 additional cases) were curated for review (50% female; 8 were children). Associated COVID-19 illnesses were febrile upper respiratory tract infections in children while adults had pneumonia (45.6%) and myocarditis (8.2%). Children had early neurologic deterioration (median day 2 in children vs day 4 in adults), seizures (5 (62.5%) children vs 3 of 9 (33.3%) adults), and motor abnormalities (6 of 7 (85.7%) children vs 3 of 7 (42.9%) adults). Eight of 12 (66.7%) adults and 4 (50.0%) children had high-risk ANE scores. Five (62.5%) children and 12 (66.7%) adults had brain lesions bilaterally and symmetrically in the putamina, external capsules, insula cortex, or medial temporal lobes, in addition to typical thalamic lesions of ANE. Hypotension was only seen in adults (30%). Hematologic derangements were common: lymphopenia (66.7%), coagulopathy (60.0%), or elevated D-dimers (100%), C-reactive protein (91.7%), and ferritin (62.5%). A pathogenic heterozygous c/.1754 C>T variant in DISCUSSION: Children and adults with SARS-CoV-2 ANE have similar clinical features and neuroimaging characteristics. Mortality is high, predominantly in patients not receiving immunotherapy and at the extremes of age.
Indexed as
Brain DiseasesCOVID-19AdultAgedChildChild, PreschoolFemaleHumansInfantMaleMethylprednisoloneMiddle AgedSARS-CoV-2SeizuresMethylprednisolone
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